Establishing core outcome domains in pediatric kidney disease: report of the Standardized Outcomes in Nephrology—Children and Adolescents (SONG-KIDS) consensus workshops

doi:10.1016/j.kint.2020.05.054

Kidney International

Volume 98, Issue 3, September 2020, Pages 553-565

https://doi.org/10.1016/j.kint.2020.05.054 Get rights and content

Trials in children with chronic kidney disease do not consistently report outcomes that are critically important to patients and caregivers. This can diminish the relevance and reliability of evidence for decision making, limiting the implementation of results into practice and policy. As part of the Standardized Outcomes in Nephrology—Children and Adolescents (SONG-Kids) initiative, we convened 2 consensus workshops in San Diego, California (7 patients, 24 caregivers, 43 health professionals) and Melbourne, Australia (7 patients, 23 caregivers, 49 health professionals). This report summarizes the discussions on the identification and implementation of the SONG-Kids core outcomes set. Four themes were identified; survival and life participation are common high priority goals, capturing the whole child and family, ensuring broad relevance across the patient journey, and requiring feasible and valid measures. Stakeholders supported the inclusion of mortality, infection, life participation, and kidney function as the core outcomes domains for children with chronic kidney disease.

Section snippets

Overview and context

Two SONG-Kids consensus workshops were convened. The first workshop was held on October 27, 2018, in San Diego, California, during the American Society of Nephrology Kidney Week, and the second workshop was held on April 13, 2019, in Melbourne, Australia, during The International Society of Nephrology World Congress of Nephrology. The SONG-Kids consensus workshops were convened to elicit stakeholder feedback on the identification and implementation of a potential core outcomes set for trials in

Overview

From the discussions across both workshops, we identified 4 themes relating to establishing a core outcomes set for trials in children with CKD: survival and life participation as common high priority goals; capturing the whole child and family; ensuring broad relevance across the patient journey; and requiring feasible and valid measures. The respective subthemes are described in the following section. Illustrative quotations for each theme are shown in Table 1. The SONG-Kids core outcomes

Postworkshop Consultation

All participants received a draft workshop report and were asked to provide feedback within a 2-week time frame. The SONG-Kids core outcomes set (Figure 1) was sent to all participants for review and comment. We uploaded the core outcomes set on the SONG website for feedback (https://songinitiative.org/projects/song-kids/). Input from the workshop participants and investigators was integrated into the final report.

Discussion

Patients, caregivers, and health professionals identified survival and life participation to be of critical importance for decision making, emphasizing that these outcomes reflected the goals of care and, therefore, should be included in the core outcomes set. Life participation was thought to capture “the whole” child and incorporated multidimensional aspects of their life including medical, lifestyle, mental health, social functioning, independence, and development or “growing well” in a

Disclosure

All the authors declared no competing interests.

Acknowledgments

The study was funded by The University of Sydney Research Accelerator Grant and the National Health and Medical Research Council (NHMRC) Program Grant Better Evidence and Translation in Chronic Kidney Disease (BEAT-CKD) (ID 1092957). CSH is supported by the NHMRC Program (grant ID1092957). AT is supported by a NHMRC Fellowship (ID 1106716). SAC is supported by the NHMRC Postgraduate Scholarship (ID 1168994). AKV is supported by a Jacquot Research Establishment Fellowship and a Princess

References (30)

M.-E. Roumelioti et al.
Sleep and fatigue symptoms in children and adolescents with CKD: a cross-sectional analysis from the Chronic Kidney Disease in Children (CKiD) study
Am J Kidney Dis
(2010)
C.J. Wong et al.
CKiD (CKD in Children) prospective cohort study: a review of current findings
Am J Kidney Dis
(2012)
S.R. Mendley et al.
Duration of chronic kidney disease reduces attention and executive function in pediatric patients
Kidney Int
(2015)
A.J. Hamilton et al.
Psychosocial health and lifestyle behaviors in young adults receiving renal replacement therapy compared to the general population: findings from the SPEAK study
Am J Kidney Dis
(2019)
C.S. Hanson et al.
Identifying important outcomes for young people with CKD and their caregivers: a nominal group technique study
Am J Kidney Dis
(2019)
A. Tong et al.
Implementing core outcomes in kidney disease: report of the Standardized Outcomes in Nephrology (SONG) implementation workshop
Kidney Int
(2018)
K. Chen et al.
Neurocognitive and educational outcomes in children and adolescents with CKD: a systematic review and meta-analysis
Clin J Am Soc Nephrol
(2018)
S.P. McDonald et al.
Long-term survival of children with end-stage renal disease
N Engl J Med
(2004)
M.M. Mitsnefes
Cardiovascular disease in children with chronic kidney disease
J Am Soc Nephrol
(2012)
F. Dobbels et al.
Health-related quality of life, treatment adherence, symptom experience and depression in adolescent renal transplant patients
Pediatr Transplant
(2010)

L. Tjaden et al.

Children's experiences of dialysis: a systematic review of qualitative studies

Arch Dis Child

(2012)

A.C. Gerson et al.

Health-related quality of life of children with mild to moderate chronic kidney disease

Pediatrics

(2010)

T.H. Diseth et al.

Kidney transplantation in childhood: mental health and quality of life of children and caregivers

Pediatr Nephrol

(2011)

A.M. McKenna et al.

Quality of life in children with chronic kidney disease—patient and caregiver assessments

Nephrol Dial Transpl

(2006)

A. Tong et al.

Standardised Outcomes in Nephrology—Children and Adolescents (SONG-Kids): a protocol for establishing a core outcome set for children with chronic kidney disease

Trials

(2016)

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Multidisciplinary Delphi consensus on challenges and key factors for an optimal care model in chronic kidney disease
2024, Nefrologia
La enfermedad renal crónica (ERC) se asocia a una elevada morbilidad, carga y consumo de recursos, y constituye un importante desafío para los sistemas sanitarios. El objetivo de este trabajo fue analizar los modelos asistenciales de atención a estos pacientes y consensuar los factores clave que deben implementarse para un modelo óptimo de atención en la ERC, a través de una visión multidisciplinar e integradora.
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Existe un consenso sobre la necesidad de priorizar la atención de la ERC tanto a nivel institucional como social, avanzando hacia modelos óptimos de atención en ERC basados en la prevención y detección precoz de la enfermedad, así como en el seguimiento integral y coordinado de los pacientes y la formación y concienciación a todos los niveles. Los factores clave identificados constituyen una hoja de ruta implantable en las distintas comunidades autónomas (CCAA) que puede contribuir a una mejora significativa en la atención a estos pacientes.
Chronic kidney disease (CKD) is associated with high morbidity, burden, and resource utilization, and represents a major challenge for healthcare systems. The purpose of this study was to analyse the care patterns for these patients and to reach a consensus on the key factors that should be implemented for an optimal care model in CKD, through a multidisciplinary and integrative vision.
A multidisciplinary panel of professionals with experience in the field of CKD was formed, composed of an advisory committee of 15 experts and an additional panel of 44 experts. Challenges and areas for improvement across the continuum of care were identified through review of scientific evidence and individual interviews with the advisory committee. Key factors for an optimal model of care in CKD were agreed and assessed using the Rand/UCLA consensus methodology (adapted Delphi), evaluating their appropriateness and necessity.
38 key factors were identified for an optimal CKD patient care model, organised into four challenges: 1) Development of CKD management models and increased visibility of the disease, 2) Prevention, optimisation of screening, early diagnosis and registration of CKD at all levels of care, 3) Comprehensive, multidisciplinary and coordinated monitoring, ensuring therapeutic optimisation and continuity of care, and 4) Reinforcement of CKD training for health care professionals and patients. 35 key factors were assessed by the panel as adequate and clearly necessary, and of these, 14 were considered highly imperative.
There is consensus on the need to prioritise CKD care at both institutional and societal levels, moving towards optimal models of CKD care based on prevention and early detection of the disease, as well as comprehensive and coordinated patient monitoring and training and awareness-raising at all levels. The key factors identified constitute a roadmap that can be implemented in the different Autonomous Communities and contribute to a significant improvement in the patient's care.
Oral Chinese patent medicines for diabetic kidney disease: An overview of systematic reviews
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Diabetic kidney disease (DKD) has become a global public health problem with a significant disease burden. A number of Chinese patent medicines (CPMs) are widely used in the treatment of DKD in China. An overview of systematic reviews (SRs) can summarize the overall evidence of its effectiveness and safety for both practice and research.
SRs and/or meta-analyses based on randomized controlled trials (RCTs) of oral CPMs for DKD were identified. Eight databases were searched from their inception to October 2021. The AMSTAR 2 tool was applied to appraise the methodological quality of the included SRs by two authors. The Grading of Recommendations Assessment, Development, and Evaluation (GRADE) approach was used to summarize the certainty of evidence of the included reviews.
A total of 88 SRs were included, of which three were published as abstracts only (quality not assessed). The methodological quality using the AMSTAR 2 approach was assessed as "low" for five SRs and "critically low" for 80 SRs. Renal end-point events and quality of life were not reported in all SRs. Most of the reviews reported surrogate outcomes. Serum creatinine and 24 h urinary total protein were the two most commonly reported indicators, and regrettably, no high-quality evidence with GRADE assessment was found. Regarding safety, except for Tripterygium glycosides tablet which had several adverse effects, other CPMs had only mild adverse effects.
Due to the lack of end-point outcomes and high-quality evidence, the effectiveness and safety of oral CPMs for DKD remain uncertain. Future studies with more critical methodological designs are needed to provide robust evidence. A core outcome set for adults with DKD could help improve the consistency and relevance of outcomes from clinical trials.
Pediatric core outcome sets had deficiencies and lacked child and family input: A methodological review
2023, Journal of Clinical Epidemiology
The Core Outcome Set-STAndards for Development (COS-STAD), published in 2017, contains 11 standards (12 criteria) describing minimum design criteria for core outcome set (COS) development. We aimed to identify and appraise all pediatric COS published prior to COS-STAD, and assess methods of child and family involvement in their development.
This methodological review included documents that described the development of pediatric COS up to and including 2017. Reviewers independently assessed each COS against COS-STAD criteria, and methods of involvement were synthesized.
A total of 56 pediatric COS were identified, meeting a median of five COS-STAD criteria. Nearly all met criteria on COS scope specification for setting, health condition, and population; 41% met criteria for intervention. Standards were more often met for the involvement of researchers/health professionals (64%) than for patients or their representatives (29%). Few met standards for achieving COS consensus (4–23%). Methods of child and family engagement varied and were limited.
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Perspectives of Clinicians on Shared Decision Making in Pediatric CKD: A Qualitative Study
2022, American Journal of Kidney Diseases
Citation Excerpt :
All participants provided signed informed consent before the interview. The interview guide (Item S1) was developed based on literature and discussion among the investigator team.21 From June 2016 to March 2020, authors C.S.H. or A.T. (both female researchers who completed training in qualitative research) conducted 1 semistructured interview with each participant in person (at a venue preferred by the participant, eg, at conference centers or clinics), by phone, or by video conference using Zoom software.
Clinical decision-making priorities may differ among children, their parents, and their clinicians. This study describes clinicians’ perspectives on shared decision making in pediatric chronic kidney disease (CKD) and identifies opportunities to improve shared decision making and care for children with CKD and their families.
Semistructured interviews.
Fifty clinicians participated, including pediatric nephrologists, nurses, social workers, surgeons, dietitians, and psychologists involved in providing care to children with CKD. They worked at 18 hospitals and 4 university research departments across 11 countries (United States of America, Canada, Australia, People’s Republic of China, United Kingdom, Germany, France, Italy, Lithuania, New Zealand, and Singapore).
Interview transcripts were analyzed thematically.
We identified 4 themes: (1) striving to blend priorities (minimizing treatment burden, emphasizing clinical long-term risks, achieving common goals), (2) focusing on medical responsibilities (carrying decisional burden and pressure of expectations, working within system constraints, ensuring safety is foremost concern), (3) collaborating to achieve better long-term outcomes (individualizing care, creating partnerships, encouraging ownership and participation in shared decision making, sensitive to parental distress), and (4) forming cumulative knowledge (balancing reassurance and realistic expectations, building understanding around treatment, harnessing motivation for long-term goals).
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Clinicians reported striving to minimize treatment burden and working with children and their families to manage their expectations and support their decision making. However, they are challenged with system constraints and sometimes felt the pressure of being responsible for the child’s long-term outcomes. Further studies are needed to test whether support for shared decision making would promote strategies to establish and improve the quality of care for children with CKD.
Sex-Based Differences in Risk Factors and Complications of Chronic Kidney Disease
2022, Seminars in Nephrology
Citation Excerpt :
Although other studies found males have an increased mortality risk, ranging between 10% and 83%,161-163 one study found the mortality risk among CKD patients was only significantly different between sexes after propensity score matching, in which males were 2.4 times (adjusted HR, 2.4; 95% CI, 1.18-4.90) more likely to die compared with females.163 Children and adolescents with kidney disease and their carers report core concerns of survival, infection, CVD, and kidney function.171 There are sparse data on survival and infection in children with CKD who have not reached kidney failure.
Globally, females are ∼30% more likely to have pre-dialysis chronic kidney disease (CKD) than males for reasons that are not fully understood. CKD is associated with numerous adverse health outcomes which makes understanding and working to eradicating sex based disparities in CKD prevalence essential. This review maps both what is known, and what is unknown, about the way sex and gender impacts (1) the epidemiology and risk factors for CKD including age, diabetes, hypertension, obesity, smoking, and cerebrovascular disease, and (2) the complications from CKD including kidney disease progression, cardiovascular disease, CKD mineral and bone disorders, anaemia, quality-of-life, cancer and mortality. This mapping can be used to guide future research.

View all citing articles on Scopus

²⁷: SONG-Kids consensus workshops investigators are listed in the Appendix.

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Meeting ReportEstablishing core outcome domains in pediatric kidney disease: report of the Standardized Outcomes in Nephrology—Children and Adolescents (SONG-KIDS) consensus workshops

Section snippets

Overview and context

Overview

Postworkshop Consultation

Discussion

Disclosure

Acknowledgments

Am J Kidney Dis

Am J Kidney Dis

Kidney Int

Am J Kidney Dis

Am J Kidney Dis

Kidney Int

Neurocognitive and educational outcomes in children and adolescents with CKD: a systematic review and meta-analysis

Clin J Am Soc Nephrol

Long-term survival of children with end-stage renal disease

N Engl J Med

Cardiovascular disease in children with chronic kidney disease

J Am Soc Nephrol

Health-related quality of life, treatment adherence, symptom experience and depression in adolescent renal transplant patients

Pediatr Transplant

Children's experiences of dialysis: a systematic review of qualitative studies

Arch Dis Child

Health-related quality of life of children with mild to moderate chronic kidney disease

Pediatrics

Kidney transplantation in childhood: mental health and quality of life of children and caregivers

Pediatr Nephrol

Quality of life in children with chronic kidney disease—patient and caregiver assessments

Nephrol Dial Transpl

Standardised Outcomes in Nephrology—Children and Adolescents (SONG-Kids): a protocol for establishing a core outcome set for children with chronic kidney disease

Trials

Meeting Report
Establishing core outcome domains in pediatric kidney disease: report of the Standardized Outcomes in Nephrology—Children and Adolescents (SONG-KIDS) consensus workshops